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Genetic Testing in Various Neurodevelopmental Disorders Which Manifest as Cerebral Palsy: A Case Study From Iran

Purpose: Cerebral palsy (CP) is a heterogeneous permanent disorder impacting movement and posture. Investigations aimed at diagnosing this disorder are expensive and time-consuming and can eventually inconclusive. This study aimed to determine the diagnostic yield of next generation sequencing in pa...

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Autores principales: Nejabat, Marzieh, Inaloo, Soroor, Sheshdeh, Afsaneh Taghipour, Bahramjahan, Shima, Sarvestani, Fatima Masoomi, Katibeh, Pegah, Nemati, Hamid, Tabei, Seyed Mohammad Bagher, Faghihi, Mohammad Ali
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8446451/
https://www.ncbi.nlm.nih.gov/pubmed/34540776
http://dx.doi.org/10.3389/fped.2021.734946
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author Nejabat, Marzieh
Inaloo, Soroor
Sheshdeh, Afsaneh Taghipour
Bahramjahan, Shima
Sarvestani, Fatima Masoomi
Katibeh, Pegah
Nemati, Hamid
Tabei, Seyed Mohammad Bagher
Faghihi, Mohammad Ali
author_facet Nejabat, Marzieh
Inaloo, Soroor
Sheshdeh, Afsaneh Taghipour
Bahramjahan, Shima
Sarvestani, Fatima Masoomi
Katibeh, Pegah
Nemati, Hamid
Tabei, Seyed Mohammad Bagher
Faghihi, Mohammad Ali
author_sort Nejabat, Marzieh
collection PubMed
description Purpose: Cerebral palsy (CP) is a heterogeneous permanent disorder impacting movement and posture. Investigations aimed at diagnosing this disorder are expensive and time-consuming and can eventually inconclusive. This study aimed to determine the diagnostic yield of next generation sequencing in patients with atypical CP (ACP). Methods: Patient eligibility criteria included impaired motor function with onset at birth or within the first year of life, and one or more of the following conditions: severe intellectual disability, positive family history, brain imaging findings not typical for cerebral palsy, abnormal neurometabolic profile, intractable seizure, normal neuroimaging despite severe psychomotor disability, after pediatric neurologist assessment including neuroimaging and biochemical-metabolic study offered for genetic study. Results: Exome sequencing was done for 66 patients which revealed pathogenic, likely pathogenic, and variants of unknown significance in 36.2, 9, and 43.9%, respectively. We also found 10 new mutations and were able to suggest specific and personalized treatments for nine patients. We also found three different mutations with different phenotypical spectrum in one gene that have not been reported for cerebral palsy. Conclusion: An accurate history and physical examination and determination of patients with atypical cerebral palsy for doing exome sequencing result in improved genetic counseling and personalized management.
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spelling pubmed-84464512021-09-18 Genetic Testing in Various Neurodevelopmental Disorders Which Manifest as Cerebral Palsy: A Case Study From Iran Nejabat, Marzieh Inaloo, Soroor Sheshdeh, Afsaneh Taghipour Bahramjahan, Shima Sarvestani, Fatima Masoomi Katibeh, Pegah Nemati, Hamid Tabei, Seyed Mohammad Bagher Faghihi, Mohammad Ali Front Pediatr Pediatrics Purpose: Cerebral palsy (CP) is a heterogeneous permanent disorder impacting movement and posture. Investigations aimed at diagnosing this disorder are expensive and time-consuming and can eventually inconclusive. This study aimed to determine the diagnostic yield of next generation sequencing in patients with atypical CP (ACP). Methods: Patient eligibility criteria included impaired motor function with onset at birth or within the first year of life, and one or more of the following conditions: severe intellectual disability, positive family history, brain imaging findings not typical for cerebral palsy, abnormal neurometabolic profile, intractable seizure, normal neuroimaging despite severe psychomotor disability, after pediatric neurologist assessment including neuroimaging and biochemical-metabolic study offered for genetic study. Results: Exome sequencing was done for 66 patients which revealed pathogenic, likely pathogenic, and variants of unknown significance in 36.2, 9, and 43.9%, respectively. We also found 10 new mutations and were able to suggest specific and personalized treatments for nine patients. We also found three different mutations with different phenotypical spectrum in one gene that have not been reported for cerebral palsy. Conclusion: An accurate history and physical examination and determination of patients with atypical cerebral palsy for doing exome sequencing result in improved genetic counseling and personalized management. Frontiers Media S.A. 2021-09-03 /pmc/articles/PMC8446451/ /pubmed/34540776 http://dx.doi.org/10.3389/fped.2021.734946 Text en Copyright © 2021 Nejabat, Inaloo, Sheshdeh, Bahramjahan, Sarvestani, Katibeh, Nemati, Tabei and Faghihi. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Pediatrics
Nejabat, Marzieh
Inaloo, Soroor
Sheshdeh, Afsaneh Taghipour
Bahramjahan, Shima
Sarvestani, Fatima Masoomi
Katibeh, Pegah
Nemati, Hamid
Tabei, Seyed Mohammad Bagher
Faghihi, Mohammad Ali
Genetic Testing in Various Neurodevelopmental Disorders Which Manifest as Cerebral Palsy: A Case Study From Iran
title Genetic Testing in Various Neurodevelopmental Disorders Which Manifest as Cerebral Palsy: A Case Study From Iran
title_full Genetic Testing in Various Neurodevelopmental Disorders Which Manifest as Cerebral Palsy: A Case Study From Iran
title_fullStr Genetic Testing in Various Neurodevelopmental Disorders Which Manifest as Cerebral Palsy: A Case Study From Iran
title_full_unstemmed Genetic Testing in Various Neurodevelopmental Disorders Which Manifest as Cerebral Palsy: A Case Study From Iran
title_short Genetic Testing in Various Neurodevelopmental Disorders Which Manifest as Cerebral Palsy: A Case Study From Iran
title_sort genetic testing in various neurodevelopmental disorders which manifest as cerebral palsy: a case study from iran
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8446451/
https://www.ncbi.nlm.nih.gov/pubmed/34540776
http://dx.doi.org/10.3389/fped.2021.734946
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